F-box only protein 41 (FBXO41)

F-box only protein 41 (FBXO41) is a substrate-recognition component of the SCF (SKP1-CUL1-F-box protein)-type E3 ubiquitin ligase complex. It is highly enriched in neurons of the central nervous system, particularly in the cerebellum, cortex, hippocampus, and spinal cord, and is absent from non-neural tissues. FBXO41 localizes mainly to the cytoplasm and centrosomes of neurons, where it promotes neuronal migration and is crucial for maintaining proper cerebellar structure and function. Functionally, FBXO41 regulates the disassembly of neuronal primary cilia by targeting centrioles and is required for correct ciliary signaling, which impacts brain development and disease. Genetic ablation of FBXO41 in animal models leads to impaired neuronal migration, cerebellar deformities, neurodegeneration, and motor disorders such as ataxia. The protein requires both its coiled-coil and F-box domains for proper localization and function, assembling into E3 ligase complexes to facilitate ubiquitin-mediated proteolysis. There is currently no evidence that FBXO41 serves as a direct therapeutic target, biomarker, or is involved in drug safety concerns.

Other names

FBXO41Fbx41KIAA1940